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儿童恶性肿瘤侵袭性毛霉菌病:意大利儿科血液肿瘤协会感染工作组的回顾性研究。

Invasive mucormycosis in children with cancer: A retrospective study from the Infection Working Group of Italian Pediatric Hematology Oncology Association.

机构信息

Department of Pediatric Oncology and Hematology, University Hospital of Policlinico, Bari, Bari, Italy.

Clinic of Pediatric Hemato-Oncology, Department of Women's and Children's Health, University Hospital of Padova, Padova, Italy.

出版信息

Mycoses. 2019 Feb;62(2):165-170. doi: 10.1111/myc.12862. Epub 2018 Nov 13.

Abstract

BACKGROUND

Invasive mucormycosis is a rare but frequently fatal fungal disease. The acute and rapidly progressive evolution causes unfavourable outcome in 22%-59% of patients and its treatment represents a clinical challenge, especially in immunocompromised patients. Current data in paediatric oncological patients are limited.

OBJECTIVES

The infection Working Group of the Italian Association of Pediatric Hematology and Oncology (AIEOP) analysed the episodes of invasive mucormycosis occurred between 2009 and 2016.

PATIENTS

Fifteen cases of proven mucormycosis (male/female 8/7; median age 14.1 years, range 7.7-18.6) were reported after chemotherapy for acute leukaemia and lymphoma (12) and allogeneic stem cell transplantation (3). The aetiology was Rhizopus oryzae 4, Lichtheimia corymbifera 3 and Mucor spp. 8.

RESULTS

Paranasal sinus was the primary site of infection in 14/15 patients combined with orbital involvement (9), central nervous system (8), lung (4), thyroid gland and kidney (1). All patients received liposomal Amphotericin B (L-AmB) (3-10 mg/kg), with surgical debridement in 14/15 cases. Eleven patients received maintenance treatment with posaconazole (9) or isavuconazole (2). Eight out of fifteen patients (53.3%) died, after 3-6 months.

CONCLUSIONS

Mucormycosis involved mainly the sinu-orbital site and affected children >10 years. Despite aggressive treatment with high-dose L-AmB and timely surgical debridement, the mortality rate remains still high.

摘要

背景

侵袭性毛霉菌病是一种罕见但常致命的真菌感染。其急性、快速进展的病程导致 22%-59%的患者预后不良,其治疗极具挑战性,尤其是在免疫功能低下的患者中。目前儿科肿瘤患者的相关数据有限。

目的

意大利儿科血液学和肿瘤学会(AIEOP)感染工作组分析了 2009 年至 2016 年间发生的侵袭性毛霉菌病病例。

患者

报告了 15 例经化疗治疗急性白血病和淋巴瘤(12 例)和异基因造血干细胞移植(3 例)后证实的毛霉菌病(男/女 8/7;中位年龄 14.1 岁,范围 7.7-18.6)。病因学为米根霉 4 例、光孢犁头霉 3 例和毛霉属 8 例。

结果

14/15 例患者的感染原发部位为鼻旁窦,合并眼眶受累(9 例)、中枢神经系统(8 例)、肺部(4 例)、甲状腺和肾脏(1 例)。所有患者均接受了脂质体两性霉素 B(L-AmB)治疗(3-10mg/kg),14/15 例患者进行了手术清创。11 例患者接受了泊沙康唑(9 例)或伊曲康唑(2 例)维持治疗。15 例患者中有 8 例(53.3%)死亡,时间为 3-6 个月后。

结论

毛霉菌病主要侵犯鼻窦-眼眶部位,影响 10 岁以上儿童。尽管采用大剂量 L-AmB 联合及时手术清创的积极治疗,死亡率仍然很高。

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